A deficiency of lunatic fringe is associated with cystic dilation of the rete testis

K. L. Hahn, B. Beres, Megan J. Rowton, M. K. Skinner, Y. Chang, A. Rawls, Jeanne Wilson-Rawls

Research output: Contribution to journalArticle

18 Citations (Scopus)

Abstract

Lunatic fringe belongs to a family of β1-3 N-acetyltransferases that modulate the affinity of the Notch receptors for their ligands through the elongation of O-fucose moieties on their extracellular domain. A role for Notch signaling in vertebrate fertility has been predicted by the intricate expression of the Notch receptors and their ligands in the oocyte and granulosa cells of the ovary and the spermatozoa and Sertoli cells of the testis. It has been demonstrated that disruption of Notch signaling by inactivation of lunatic fringe led to infertility associated with pleiotropic defects in follicle development and meiotic maturation of oocytes. Lunatic fringe null males were found to be subfertile. Here, we report that gene expression data demonstrate that fringe and Notch signaling genes are expressed in the developing testis and the intratesticular ductal tract, predicting roles for this pathway during embryonic gonadogenesis and spermatogenesis. Spermatogenesis was not impaired in the majority of the lunatic fringe null males; however, spermatozoa were unilaterally absent in the epididymis of many mice. Histological and immunohistochemical analysis of these testes revealed the development of unilateral cystic dilation of the rete testis. Tracer dye experiments confirm a block in the connection between the rete testis and the efferent ducts. Further, the dye studies demonstrated that many lunatic fringe mutant males had partial blocks of the connection between the rete testis and the efferent ducts bilaterally.

Original languageEnglish (US)
Pages (from-to)79-93
Number of pages15
JournalReproduction
Volume137
Issue number1
DOIs
StatePublished - 2009

Fingerprint

Rete Testis
Notch Receptors
Testis
Dilatation
Spermatogenesis
Oocytes
Spermatozoa
Coloring Agents
Ligands
Fucose
Sertoli Cells
Granulosa Cells
Epididymis
Infertility
Fertility
Vertebrates
Ovary
Gene Expression
Genes

ASJC Scopus subject areas

  • Obstetrics and Gynecology
  • Reproductive Medicine
  • Embryology
  • Endocrinology
  • Cell Biology

Cite this

A deficiency of lunatic fringe is associated with cystic dilation of the rete testis. / Hahn, K. L.; Beres, B.; Rowton, Megan J.; Skinner, M. K.; Chang, Y.; Rawls, A.; Wilson-Rawls, Jeanne.

In: Reproduction, Vol. 137, No. 1, 2009, p. 79-93.

Research output: Contribution to journalArticle

Hahn, K. L. ; Beres, B. ; Rowton, Megan J. ; Skinner, M. K. ; Chang, Y. ; Rawls, A. ; Wilson-Rawls, Jeanne. / A deficiency of lunatic fringe is associated with cystic dilation of the rete testis. In: Reproduction. 2009 ; Vol. 137, No. 1. pp. 79-93.
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